Rare Genetic Variants in Complement Factor I Lead to Low FI Plasma Levels Resulting in Increased Risk of Age-Related Macular Degeneration

Invest Ophthalmol Vis Sci. 2020 Jun 3;61(6):18. doi: 10.1167/iovs.61.6.18.

Abstract

Purpose: Rare genetic variants in complement factor I (CFI) that cause low systemic levels of the protein (FI) have been reported as a strong risk factor for advanced age-related macular degeneration (AMD). This study set out to replicate these findings.

Methods: FI levels were measured by sandwich ELISA in an independent cohort of 276 patients with AMD and 205 elderly controls. Single-nucleotide polymorphism genotyping and Sanger sequencing were used to assess genetic variability.

Results: The median FI level was significantly lower in those individuals with AMD and a rare CFI variant (28.3 µg/mL) compared to those with AMD without a rare CFI variant (38.8 µg/mL, P = 0.004) or the control population with (41.7 µg/mL, P = 0.0085) or without (41.5 µg/mL, P < 0.0001) a rare CFI variant. Thirty-six percent of patients with AMD with a rare CFI variant had levels below the fifth percentile, compared to 6% in controls with CFI variants. Multiple regression analyses revealed a decreased FI level associated with a rare CFI variant was a risk factor for AMD (early or late AMD: odds ratio [OR] 12.05, P = 0.03; early AMD: OR 30.3, P = 0.02; late AMD: OR 10.64, P < 0.01). Additionally, measurement of FI in aqueous humor revealed a large FI concentration gradient between systemic circulation and the eye (∼286-fold).

Conclusions: Rare genetic variants in CFI causing low systemic FI levels are strongly associated with AMD. The impermeability of the Bruch's membrane to FI will have implications for therapeutic replacement of FI in individuals with CFI variants and low FI levels at risk of AMD.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Aged, 80 and over
  • Biomarkers
  • Case-Control Studies
  • Complement Factor H / genetics
  • Complement Factor I / genetics*
  • Complement Factor I / metabolism
  • Eye Proteins / genetics*
  • Female
  • Genetic Predisposition to Disease / genetics*
  • Genetic Testing
  • Genetic Variation*
  • Genotyping Techniques
  • Humans
  • Macular Degeneration / blood
  • Macular Degeneration / genetics*
  • Male
  • Middle Aged
  • Polymorphism, Single Nucleotide*
  • Proportional Hazards Models
  • Proteins / genetics
  • Receptors, TNF-Related Apoptosis-Inducing Ligand / genetics
  • Retrospective Studies
  • Risk Factors
  • Vascular Endothelial Growth Factor A / genetics

Substances

  • ARMS2 protein, human
  • Biomarkers
  • CFH protein, human
  • Eye Proteins
  • Proteins
  • Receptors, TNF-Related Apoptosis-Inducing Ligand
  • TNFRSF10A protein, human
  • VEGFA protein, human
  • Vascular Endothelial Growth Factor A
  • Complement Factor H
  • CFI protein, human
  • Complement Factor I